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Case Report High Fever As an Initial Symptom of Primary Gastric Inflammatory Myofibroblastic Tumor in an Adult Woman

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Case Report High Fever As an Initial Symptom of Primary Gastric Inflammatory Myofibroblastic Tumor in an Adult Woman Int J Clin Exp Med 2014;7(5):1468-1473 www.ijcem.com /ISSN:1940-5901/IJCEM0000684 Case Report High fever as an initial symptom of primary gastric inflammatory myofibroblastic tumor in an adult woman Jiang-Feng Qiu, Yi-Jiu Shi, Lei Fang, Hui-Fang Wang, Mou-Cheng Zhang Department of Gastrointestinal Surgery, Ningbo First Hospital, Ningbo, 315010, China Received March 29, 2014; Accepted May 9, 2014; Epub May 15, 2014; Published May 30, 2014 Abstract: Inflammatory myofibroblastic tumor, also known as inflammatory pseudotumor, plasma cell granuloma or inflammatory myofibroblastoma, is characterized histopathologically by myofibroblastic spindle cells with inflamma- tory cell infiltrates composed of plasma cells, lymphocytes and eosinophils. Inflammatory myofibroblastic tumor is typically seen in children or young adults and is most commonly localized to the lungs, but it can occur anywhere in the body. To date, however, only a few cases involving the stomach have been reported. Herein, we present a case of gastric inflammatory myofibroblastic tumor in an adult woman with an initial symptom of high fever. Keywords: Inflammatory myofibroblastic tumor, stomach, inflammatory pseudotumor, high fever, surgery Introduction tenderness. Routine blood tests revealed mi- crocytic hypochromic anemia with a hemoglo- Inflammatory myofibroblastic tumor (IMT) is an bin level of 10.8 g/dl and a hematocrit of 34.3%. uncommon mesenchymal neoplasm occurring Repeated blood cultures came up negative for mainly in children and young adults. IMT was the presence of bacteria or fungus. Radio- first described in the lung, but has since been logically, chest X-rays were normal, but con- observed in a wide variety of extrapulmonary trast-enhanced abdominal computed tomogra- sites such as the liver, urinary bladder, mesen- phy (CT) showed a 3.0 × 3.0 cm low-density tery, retroperitoneum, omentum and central mass located on the lesser curvature of the nervous system [1]. IMT is rarely seen in the stomach, near the angular incisure, and with stomach, especially in adults [2]. Here we several enlarged lymph nodes around it (Figure report a case of primary gastric IMT in an adult 1). Flexible upper digestive endoscopy identi- woman with high fever as the initial symptom. fied a small, superficial mucosal lesion in the The clinical and histopathological features of gastric antrum, and endoscopic ultrasound this rare lesion are described with a review of (EUS) showed a round hypoechoic mass, 3.0 × the literature. 3.0 cm in diameter, originating from the muscu- laris propria layer (Figure 2). Endoscopic biopsy Case presentation revealed inflamed gastric mucosa. Upon admis- A 61-year-old female was admitted to the emer- sion, the patient was initially administered intra- gency department suffering from a high fever of venous piperacillin and tazobactam for 3 days, 39.8°C. She had been experiencing intermit- followed by 3 days of intravenous meropenem, tent epigastric pain and abdominal distention but her temperature continued to fluctuate over a period of 5 days before fever developed, between 39°C and 39.8°C. The patient’s tem- but had no other presenting symptoms such as perature finally returned to normal after being cough, nausea, vomiting, hematochezia, reflux, treated with dexamethasone and indome- asthma or shivering. In addition, she had no thacin. history of alteration in bowel habits. Her past medical history was unremarkable, with the Based on the findings described above, a diag- exception of hypertension for the past 10 years. nosis of gastric sarcoma was suspected, and surgical excision was recommended due to the Clinical examination did not reveal any palpable presence of a gastric mass with unclear tumor abdominal mass and there were no signs of histology. The patient therefore underwent Gastric inflammatory myofibroblastic tumor in a woman Figure 1. Abdomen CT scan. CT scan shows a 3.0 × 3.0 cm low-density mass, marked with the arrow, located on the lesser curvature near the angular incision. Figure 2. Flexible upper GI endoscopy. A small, superficial mucosal lesion marked with the arrow in the gastric antrum is demonstrated (A). EUS shows a round hypoechoic mass, 3.0 × 3.0 cm in diameter, originating from the muscularis propria layer (B). CD35, IGg4 and anaplastic lymphoma kinase exploratory laparotomy 10 days after admis- (ALK) (Figure 3). The immunological and mor- sion, during which the tumor’s location in the phological findings were found to be consistent lesser curvature near the angular incisure was with the diagnosis of IMT in the present case. confirmed, where it was found to have infiltrat- The postoperative course was uneventful, and ed the entire thickness of the gastric wall. the patient appears to be doing well over 3 Several enlarged lymph nodes were observed months of follow-up. near the tumor, but no signs of adjacent organ invasion or metastasis were detected. A radical Discussion surgical procedure was performed including a distal gastrectomy with D2 lymph node dissec- IMT has been characterized as a histologically tion and Billroth I reconstruction. Histopa- distinctive lesion with unpredictable behavior. thological examination of the tumor revealed While there was previous debate as to whether spindle cells with inflammatory infiltrate of neu- IMT is a tumor or inflammation, it is now recog- trophils, eosinophils, lymphocytes and plasma nized as a neoplasm of intermediate biological cells. Immunohistochemical analysis showed potential, with frequent recurrences and only that the tumor cells were positive for vimentin occasional metastasis [3]. The etiopathogene- and smooth muscle actin (SMA), but negative sis of IMT remains unclear. Various mecha- for cytokeratin (CK), CD117, CD21, CD23, nisms of tumor development have been postu- 1469 Int J Clin Exp Med 2014;7(5):1468-1473 Gastric inflammatory myofibroblastic tumor in a woman Figure 3. Microscopy of gastric IMT. Image shows spindle cells with inflammatory cell infiltration (A). Immunohisto- chemistry reveals the presence of vimentin (B) and smooth muscle actin (SMA) (C); whereas negative for ALK (D), CD117 (E), CK (F), CD21 (G), CD23 (H), CD35 (I) and IGg4 (J) (HE staining ×10). lated, including exaggerated inflammatory tumor (GIST), leiomyoma and follicular dendritic response to infections and rearrangement of cell sarcoma (FDCS) that must be distinguished the ALK gene [4, 5]. As stated previously, it from IMT. Usually, IFP, GIST and smooth muscle occurs mainly in children and young adults, and tumors do not display the systemic symptoms the lungs are the most commonly affected site. as seen in IMT patients. The definitive diagno- Primary gastric IMTs in adults are very rare. To sis should be verified by a histopathological date, only 16 adult patients with gastric IMT study. IFP is a benign tumor-like lesion which have been reported based on the available lit- rarely invades the muscularis. The tumor cells erature, with a male to female ratio of 1 to 4 [6, in IFP also present an onion skin-like pattern 7]. Clinical and pathological findings from a around blood vessels and glands which is review of 16 cases of gastric IMT in adults that absent in IMT. Immunohistochemically, IFP has been reported are summarized in Table 1. mostly presents positive for CD34 but negative for SMA [17]. In the present case, IFP was Gastric IMT typically present with abdominal therefore ruled out by the transmural location, pain, hematemesis, melena and a palpable lack of onion skinning on histology and immu- abdominal mass. Weight loss, fever and hypo- nopositivity for SMA. GIST microscopically chromic anemia may also be associated with shows only scattered inflammatory cells, and gastric IMT. To our knowledge, however, the immunohistochemically nearly always presents present case is the first report of primary gas- positive for CD117 and DOG1 which was oppo- tric IMT in adults with high fever of almost 40°C site for IMT [18]. Although leiomyoma can show as the initial and main symptom. myxoid change and occasional eosinophilic infiltrates with immunohistochemically positive Radiologically, there is no specific feature to the for SMA, leiomyomas do not demonstrate IMT [16]. Pre-surgical EUS and CT may help to marked lymphoplasmacytic infiltrates and determine the extragastric invasion. Due to the sparsely arranged spindle cells, moreover, leio- tumor’s submucosal location, endoscopic biop- myomas often show numerous scattered sies frequently reveal only normal gastric CD117 positive mast cells, which helped in dif- mucosa. Surgical exploration with complete ferentiating it from IMT [7]. Follicular dendritic resection is therefore the most efficient treat- cell sarcoma is a malignant neoplasm derived ment method and can meanwhile obtain an from follicular dendritic cell that possess and accurate histological diagnosis. The histologi- present antigens to B cells in the follicular cen- cal appearance of gastric IMT is characterized ters of lymphoid organs. They often occur in by proliferation of spindle-shaped myofibro- lymph nodes, although they can also arise at blastic cells and a lymphoplasmacytic infiltrate extranodal sites. Histologically, the characteris- distributed among the tumor cells. Immu- tics of FDCS are whorl, storiform and fascicular nohistologically, IMT cells are usually positive arrangements of oval to spindle tumor cells for vimentin, SMA, desmin, but negative for with indistinct cell borders. Immunohisto- CD117 and S-100. There are a few tumors or chemically, the most sensitive and specific lesions in the stomach such as inflammatory markers for FDCS are positive for CD21, CD23 fibroid polyp (IFP), gastrointestinal stromal and CD35 but negative for CK which may be 1470 Int J Clin Exp Med 2014;7(5):1468-1473 Gastric inflammatory myofibroblastic tumor in a woman Table 1. Review of patient characteristics of previously reported gastric IMT Age/ Tumor ALK Author Presenting symptoms Tumor location Treatment Follow-up sex size status 1 Kojimahara et al. 1993 [8] 19/F Vomiting, Weight loss C to LC 9 cm TG NA Asymptomatic at 2.5 years 2 Al-Taie et al.
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